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Objective: The aim of this systematic review was to identify patient-reported outcome (PRO) instruments used in research with children/youth with conditions associated with facial differences to identify the health concepts measured., Design: MEDLINE, EMBASE, CINAHL, and PsycINFO were searched from 2004 to 2016 to identify PRO instruments used in acne vulgaris, birthmarks, burns, ear anomalies, facial asymmetries, and facial paralysis patients. We performed a content analysis whereby the items were coded to identify concepts and categorized as positive or negative content or phrasing., Results: A total of 7,835 articles were screened; 6 generic and 11 condition-specific PRO instruments were used in 96 publications. Condition-specific instruments were for acne (four), oral health (two), dermatology (one), facial asymmetries (two), microtia (one), and burns (one). The PRO instruments provided 554 items (295 generic; 259 condition specific) that were sorted into 4 domains, 11 subdomains, and 91 health concepts. The most common domain was psychological (n = 224 items). Of the identified items, 76% had negative content or phrasing (e.g., "Because of the way my face looks I wish I had never been born"). Given the small number of items measuring facial appearance (n = 19) and function (n = 22), the PRO instruments reviewed lacked content validity for patients whose condition impacted facial function and/or appearance., Conclusions: Treatments can change facial appearance and function. This review draws attention to a problem with content validity in existing PRO instruments. Our team is now developing a new PRO instrument called FACE-Q Kids to address this problem.

Objective: Lip asymmetry after a unilateral cleft lip repair can be perceived as an unsatisfactory result. The objective of this study is to determine the degree of upper lip asymmetry and/or nasal alar hooding required for recognition of asymmetry in a simulated model of unilateral cleft lip., Design: A model of unilateral cleft lip was created using digital morphing software to simulate asymmetries in vermilion height and nasal hooding in photographs of children. Volunteers were shown photographs for different time intervals and with varying degrees of asymmetry. Ability to detect facial asymmetry was recorded and analyzed., Setting: This study was conducted by surveying layperson volunteers in public community settings., Participants: 108 layperson volunteers were randomly surveyed., Main Outcome Measures: The primary outcome measure was a reported lip or nose asymmetry by the volunteers. Proportions and corresponding 95% confidence intervals were obtained to estimate the probability of reporting an asymmetry at 3- and 10-second intervals., Results: After 3- and 10-second exposure, labial asymmetry was perceived by ≥50% of subjects at 2 mm (62%, P = .001) and 1 mm (89%, P < .0001), respectively. Nasal asymmetry was detected by <50% of subjects at 3 seconds, but ≥50% perceived a 3-mm alteration at 10 seconds (64%, P < .0001). Photographs with combined nasal and labial modification did not lower the threshold for asymmetry perception compared to either deformity alone., Conclusions: This study is the first to determine a predictable millimeter threshold for perceived asymmetry in cleft lip deformity using a digital model.

Objective: To assess how faces with a cleft lip and palate are perceived and to study how faces with and without a unilateral cleft lip and palate are viewed by individuals with a unilateral cleft lip and/or palate in comparison to nonaffected controls., Design: Prospective clinical study., Setting: Department of Orthodontics and Department of Psychology, University of Wuerzburg., Participants: Thirty-three participants (20 men and 13 women; mean age, 25.4 ± 6.6 years) with a unilateral cleft lip and/or palate and a control group of 30 participants (15 men and 15 women; mean age, 26.8 ± 3.4 years) were enrolled in this study., Main Outcome Measures: Eye movements were analyzed via an eye-tracking camera while all participants looked at pictures of faces with and without a unilateral cleft lip and palate., Results: The nose and the mouth area of pictures of faces with a unilateral cleft lip and palate were looked at significantly longer by both groups. Additionally, the participants with a unilateral cleft lip and/or palate looked at faces with and without a unilateral cleft lip and palate differently, taking more time to view the nose and less time to view the eyes compared with the participants without a cleft lip and palate., Conclusion: When perceiving a face with a unilateral cleft lip and palate, the observer's gaze is distracted to the nose and mouth area. Moreover, participants with a unilateral cleft lip and/or palate themselves focused greater attention on those features that are anomalous on their own faces in comparison to participants without a cleft lip and palate. Specifically, this different scanpath is reflected in the cumulative duration of the eye movements as well as in the initial facial scan pattern.

Objective: To obtain descriptive information about diagnosis-specific patterns of psychosocial adjustment for children and adolescents with craniofacial anomalies., Design: Chart review of medical records was used to obtain psychosocial checklists. Scores were compared with published norms to evaluate elevated risk of problems, separately for six diagnostic groups., Setting: Plastic surgery department in urban university medical center., Participants: As part of routine ongoing care, 408 caregivers completed checklists assessing psychosocial adjustment of children aged 2 to 18 years with diagnosis of cleft lip and palate, cleft lip only, cleft palate only, craniosynostosis, hemifacial microsomia, and hemangioma., Main Outcome Measure: Child Behavior Checklist ( Achenbach, 1991 , 1992 )., Results: The craniosynostosis group showed elevated rates (beyond norms) of social, academic, and attention problems. The cleft lip and palate group showed elevations in social problems and deficits in social, academic, and activities competencies. The other diagnostic groups had few areas of elevated problems and had reduced risk in some areas., Conclusion: Specific patterns of strengths and weaknesses in psychosocial adjustment may vary by craniofacial anomaly diagnosis. Replication of these findings, with extension to other craniofacial anomaly diagnoses, is warranted.

Objective: Evaluation of the Dutch Child Oral Health Impact Profile (COHIP), assessing the level of concordance between parents and children. The internal consistency and the predictive validity of the COHIP for self-reported general health were examined., Methods: Sample size was 35 pairs of parents and children age 11 to 14 with craniofacial conditions. Cronbach alphas were calculated and the level of concordance between parents and children was studied using t tests and intraclass correlations. Predictive validity was assessed using Pearson correlations and linear regression analyses., Results: The COHIP and its subscales, except for one, had satisfactory to high Cronbach alphas (.59 to .94). Parents and children did not differ significantly. Correlations between parents and children were high (.62 to .91). Only "Oral symptoms" proved to be a significant predictor of general health, but only in the parent sample., Conclusions: In spite of the high level of concordance found, proxy reports have to be considered complementary to the reports of the children themselves. The Dutch version of the COHIP performs adequately, but could use some further psychometric evaluation and revision. It does not seem advisable to use the subscales separately as predictors in the same regression model, since they are strongly intercorrelated. For use in craniofacial patients, further validation is needed on a larger sample and some items need to be revised or removed. Finally, given the small number of cases, conclusions must be drawn with caution.

Objective: Previous psychosocial studies of adults born with cleft lip and palate have provided circumstantial evidence that surgically repaired right-sided unilateral clefts may be more disfiguring than left-sided clefts. The present study asked if such asymmetries are physiognomic asymmetries or arise "in the eye of the beholder," representing perceptual processes in face recognition., Design: Color slides of 160 children (6 years of age) and young teenagers (16 years of age) were rated by subjects for perceived disfigurement. Sixty of the subjects had unilateral complete cleft lip and palate (30 had a right-sided cleft and 30 had a left-sided cleft), 60 had unilateral cleft lip/alveolus (30 right-sided and 30 left-sided clefts), 32 children had bilateral cleft lip and palate, and 8 children had cleft palate only. Faces were shown in normal and in mirror-reversed versions; the order in which faces were shown was randomized, as were other stimulus factors such as cleft type, age, and gender., Setting: The study was conducted as a classroom-type experiment at the Vision Laboratory, Department of Psychology, Oslo, Norway., Participants: Thirty-seven students of psychology at the University of Oslo, who were ignorant of the purpose of the study, acted as subjects., Main Outcome Measure: Subjects rated perceived disfigurement using a visual analog scale., Results: Modest but highly consistent hemifacial asymmetries in judged disfigurement were found, with left-sided unilateral clefts rated as less disfiguring than right-sided unilateral clefts. Unilateral clefts were judged as being less disfiguring than the bilateral clefts, and cleft lip/alveolus was judged as being less disfiguring than cleft lip and palate. The patterns of facial judgments were almost identical in the normal and reversed-slides conditions., Conclusions: Asymmetries between left- and right-sided clefts reside in physiognomic factors rather than in hemispheric asymmetries controlling the perceptual process of face judgment.

Objective: To compare the psychosocial adjustment of children with and without hemifacial microsomia (HFM)., Design: This cross-sectional pilot study investigated psychosocial adjustment of twin pairs in which one twin was affected with HFM and the other was unimpaired. Data were analyzed using paired t tests., Setting: The dental medicine clinic of a large urban children's hospital, serving a multistate area., Participants: Participants were six twin pairs aged 9 to 15 years and their parents. Data were also collected from classroom teachers., Main Outcome Measures: Outcome measures included: the Child Behavior Checklist (CBCL), the Teacher Report Form (TRF), the Coopersmith Self-Esteem Inventory (SEI), and a structured self-concept interview (the Self-Interview)., Results: Children with HFM had significantly higher CBCL and TRF total behavior problem scores and lower SEI general self-esteem scores than their unimpaired twins. No differences were found in CBCL competence scores. Interview data revealed concerns among the HFM group related to appearance, negative social responses, and fear of hospital procedures. Children also reported positive aspects of coping with HFM., Conclusions: The results of this study suggest that, similar to other craniofacial conditions, HFM is associated with an elevated risk for childhood psychosocial difficulties. Replication of specific findings with a larger sample is needed. Multicenter, collaborative studies are a crucial next step for this field.

Psychosocial adjustment was evaluated in different groups of children with craniofacial deformities in order to examine the relationships between symmetry, functional impairments, and social adaptation. Thirty patients, ages 6 through 16 years, were assessed using a battery of standard psychologic measures including Human Figure Drawing, Tasks of Emotional Adjustment, and Children's Depression Inventory. Parents and teachers also provided ratings of the children's functioning using the Child Behavior Checklist (parent and teacher forms). Scores on the outcome measures were summarized with descriptive statistics and then multivariate analyses of variance were conducted to determine the differences in psychosocial development between children with symmetric versus asymmetric craniofacial deformities and between the presence or absence of a functional impairment in children with craniofacial deformities. The results indicate that children with symmetric craniofacial deformities score poorer on measures of psychosocial adjustment than children with asymmetric deformities (F = 3.20, p less than 0.015); however, differences among the groups with or without functional impairments were not significant.